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ATTACH = Anti-TNF-α Therapy Against Chronic Heart failure; CHF = congestive heart failure; CVD = cardiovascular disease; IL = interleukin; LV = left ventricular; LVEF = left ventricular

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ATTACH = Anti-TNF-α Therapy Against Chronic Heart failure; CHF = congestive heart failure; CVD = cardiovascular disease; IL = interleukin; LV = left ventricular; LVEF = left ventricular ejection fraction; MI = myocardial infarction; MMP = matrix metalloproteinase; OA = osteoarthritis; RA = rheumatoid arthritis; RECOVER = Research into Etanercept: Cytokine Antagonism in Ventricular Dysfunction Trial; RENNAISANCE = Randomized Etanercept North American Strategy to Study Antagonism of Cytokines; RF rheumatoid factor; TACE = TNF-α converting enzyme; TIMP = tissue inhibitor of matrix metalloproteinase; TNF-α = tumor necrosis factor-α

Abstract

Data from population- and clinic-based epidemiologic studies of

rheumatoid arthritis patients suggest that individuals with rheumatoid

arthritis are at risk for developing clinically evident congestive heart

failure Many established risk factors for congestive heart failure are

over-represented in rheumatoid arthritis and likely account for some

of the increased risk observed In particular, data from animal models

of cytokine-induced congestive heart failure have implicated the

same inflammatory cytokines produced in abundance by rheumatoid

synovium as the driving force behind maladaptive processes in the

myocardium leading to congestive heart failure At present, however,

the direct effects of inflammatory cytokines (and rheumatoid arthritis

therapies) on the myocardia of rheumatoid arthritis patients are

incompletely understood

Introduction

Unique cardiac complications of rheumatoid arthritis (RA),

such as cardiac rheumatoid nodules, have been recognized

for over a century It has only been appreciated in the last

decades, however, that certain chronic autoimmune

inflam-matory diseases, such as RA and systemic lupus

erythema-tosis, increase the risk of developing cardiovascular disease

(CVD), particularly atherosclerosis and congestive heart

failure (CHF) [1-5] In fact, striking commonalities in the

cellular and cytokine profiles of the rheumatoid synovial lesion

and atherosclerotic plaque [6-8] have prompted speculation

that the inflammatory pathways of RA may initiate and/or

accelerate plaque formation and that this effect may be

ameliorated by anti-inflammatory therapies [9]

The link between RA and CHF is less well studied The CHF

phenotype can evolve from a variety of pathogenic conditions,

many of which may be promoted by the RA disease process

Yet to date, only a handful of investigations have attempted to

dissect this complex issue A particular source of confusion has been the apparent contradiction between pre-clinical studies linking inflammation to CHF and the lack of efficacy of anti-cytokine therapy in clinical trials in advanced CHF (discussed below) Because anti-cytokine therapy has become a cornerstone in the treatment of RA, it is particularly critical to understand the contribution of cytokine-induced inflammation to myocardial structure and function in RA Here, we review the current literature on the epidemiology of CHF in RA with an emphasis on the pathogenesis of cytokine induced myocardial dysfunction

Epidemiology of congestive heart failure:

general considerations

The epidemiology of CHF in RA, and the limitations of the available data, are better appreciated in the context of estimates of CHF in the general population The prevalence

of CHF in western countries appears to have been increasing over the past few decades, due primarily to increased longevity rather than to a change in incidence rates [10] In the United States, more than 400,000 new cases of CHF are identified each year and added to the estimated 2.5 to

5 million Americans with prevalent CHF [11,12], yielding an overall prevalence of 1.1% to 2% of the population Nearly 300,000 deaths in the US are attributed to CHF annually [10] For persons over the age of 65 years, CHF is the most frequent cause of hospitalization [11,13]

Incidence rates of CHF vary among published reports, presumably reflecting differences in the populations studied, diagnostic criteria used, and temporal trends in coding practices for reimbursement [14] Recent data from several community-based cohorts [15-18] have yielded an estimated

Review

Myocardial dysfunction in rheumatoid arthritis: epidemiology

and pathogenesis

Jon T Giles1, Verônica Fernandes2, Joao AC Lima2and Joan M Bathon1

1Division of Rheumatology, The Johns Hopkins University School of Medicine, Baltimore, MD, USA

2Division of Cardiology, The Johns Hopkins University School of Medicine, Baltimore, MD, USA

Corresponding author: Jon T Giles, gilesjont@jhmi.edu

Published: 24 August 2005 Arthritis Research & Therapy 2005, 7:195-207 (DOI 10.1186/ar1814)

This article is online at http://arthritis-research.com/content/7/5/195

© 2005 BioMed Central Ltd

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age-adjusted incidence of CHF of 3.4 to 17.6 per 1,000

person-years for men and 2.4 to 12.5 per 1,000 person-years

for women The wide range in rates reflects, at least in part,

differences in diagnostic criteria used from study to study For

example, age-adjusted incidence rates based on the

Framingham diagnostic criteria for heart failure [15,16,18,19]

(Table 1) were between 2 and 4 per 1,000 person-years,

whereas rates based on less stringent criteria were three- to

four-fold higher [17]

The incidence of CHF increases with age [20,21]; 88% of

affected individuals are over the age of 65 years, and 49% are

over 80 years at diagnosis [20] The remaining lifetime risk of

developing CHF at all index ages from 40 through 80 years of

age is between 20% and 33%, and is roughly equal for men

and women [17,22] Levy et al [15] have shown that over the

past 50 years the incidence of CHF has declined among

women but not among men This lack of decline in CHF

incidence among men is largely attributable to advances in the

management of acute myocardial infarction, diabetes, and

hypertension that have led to an overall decrease in mortality

rates from these disorders while adding to the incidence of

CHF [23] Survival after the onset of CHF has improved in

both sexes [15] Factors contributing to the decrease in CHF

mortality include improved access to care, the introduction of

effective therapies, and improved care of comorbid conditions

[15] Despite these encouraging trends, mortality rates of

patients with CHF remain alarmingly high Recent reports from

community-based cohorts [15-18] estimate age-adjusted

one-and five-year CHF mortality at 23% to 27% one-and 45% to 65%, respectively For women in these series, survival was slightly better than [15,16] or equal to [17,18] men

Epidemiology of congestive heart failure in rheumatoid arthritis

Fewer statistics on incidence and prevalence rates for CHF in patients with RA are available and are derived from a handful

of population-based [24-26] and clinic-based RA cohorts

[5,27,28] Gabriel et al [24] estimated the incidence of CHF

among all RA patients in Olmsted County, Minnesota, from data abstracted from medical records Between 1955 and

1985, 78 cases of incident CHF were identified among 450 prevalent cases of RA compared to 54 cases among the same number of non-RA community controls matched for age, sex, and baseline comorbidity, yielding a relative risk of 1.60 (95% CI 1.12-2.27) In contrast, the risk of incident CHF in patients with osteoarthritis (OA), a non-inflammatory arthritis, was not increased compared to non-OA community controls [24] In a follow-up retrospective review of the same cohort extended to 1995, now using the Framingham

diagnostic criteria for CHF (Table 1), Nicola et al [26]

confirmed an increased risk of incident CHF in both rheumatoid factor (RF) negative and positive RA patients (hazards ratio 1.34 and 2.29, respectively) compared to

non-RA controls adjusted for age, sex, and CV risk factors Incident CHF risk remained elevated after further adjustment for comorbid ischemic heart disease (hazards ratio 1.28 and 2.59 for RF negative and positive RA patients, respectively), although the risk relationship was no longer statistically significant for RF negative patients in this model [26]

In a combined cohort of RA patients from community-based

practices and drug safety monitoring studies (n = 9093), Wolfe et al [5] estimated an adjusted lifetime relative risk of

CHF in patients with RA of 1.43 (95% CI 1.24-1.33) compared with OA controls The adjusted lifetime prevalence

of CHF in the RA population was 2.34% compared to 1.64%

in OA controls Data were collected via patient survey of self-reported, physician-diagnosed CHF, and confirmed by review

of a random sample of medical records in 50% of patients reporting CVD events In a subsequent analysis [27], in which

the drug safety cohort represented a third (n = 4,307) of the total sample (n = 13,171), Wolfe et al reported an adjusted

frequency of CHF of 3.9% (95% CI 3.4-4.3%) in RA patients compared to 2.3% (95% CI 1.6-3.3%) in controls with knee

or hip OA Factors associated with prevalent and incident CHF were those typically associated with CHF in the non-RA population (e.g., age, male gender, hypertension, coronary artery disease, diabetes, and smoking) while RA-related measures (patient-reported disability, pain, and RA global severity) were also associated with prevalent and incident CHF As data were collected by mailed questionnaire, objective measures of RA disease activity (e.g., swollen and tender joint counts and serum inflammatory markers) were not available to assess as predictor variables

Table 1

Framingham diagnostic criteria for congestive heart failure [19]

Major criteria

Paroxysmal nocturnal dyspnea

Neck vein distension

Pulmonary rales

Radiographic cardiomegaly (chest radiography)

Acute pulmonary edema

Third heart sound gallop

Central venous pressure > 16 cm water

Circulation time ≥ 25 seconds

Hepatojugular reflux

Weight loss ≥ 4.5 kg in 5 days in response to treatment with diuretics

Paroxysmal nocturnal dyspnea

Minor criteria

Bilateral ankle edema

Nocturnal cough

Dyspnea on ordinary exertion

Hepatomegaly

Pleural effusion

Decrease in vital capacity by 33% of maximum

Heart rate ≥ 120 beats per minute

Bilateral ankle edema

Nocturnal cough

Two major or one major and two minor criteria are required for a

clinical diagnosis of CHF

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Indeed, the impact of CHF in RA may be under-appreciated

due to excess CHF related mortality Mutru et al [28] first

reported a higher rate of CHF-attributed mortality in RA

patients compared to age- and gender-matched controls with

CHF for both males (P = 0.004) and females (P = 0.042) In

a recent report, Nicola et al [29] found that CHF preceded

nearly two-thirds of the excess CVD associated deaths in RA

patients compared to age- and gender-matched controls

These unexpected results alone emphasize a need for greater

understanding of the dynamics of myocardial dysfunction in

RA and suggest that survivor-bias may serve to

underestimate the true extent of CHF in the RA population

An important limitation of each of these studies is in the

method chosen to ascertain the diagnosis of CHF The

application of clinical criteria alone for the diagnosis of CHF

is too imprecise, as demonstrated by one study [30] in which

a false positive diagnosis of CHF was made by primary care

providers in over one-third of patients Current guidelines

[10] advocate the need for Doppler echocardiographic

confirmation of any diagnosis of CHF when suspected

clinically Reliance on clinical diagnostic criteria alone may

result in over-diagnosis of CHF in some case In others, CHF

may be under-diagnosed when dependent ankle edema is

mistaken for joint swelling, chronic pulmonary congestion is

misinterpreted as rheumatoid lung involvement, and exertional

dyspnea is masked by a sedentary lifestyle due to painful joint

deformities Nevertheless, on balance, the available data

support higher prevalence and incidence rates of CHF in RA

patients compared to matched controls without RA Many

factors unique to or over-represented in RA patients may

explain, at least in part, why the myocardium is at risk in RA In

addition, an analysis of the relative contributions of each of

these risk factors and associated biomarkers to the

development of CHF in RA patients invites speculation into

the underlying pathophysiologic mechanisms leading to

myocardial dysfunction

Risk factors, echocardiographic predictors,

and biochemical markers associated with the

development of CHF: relationship to RA

Risk factors for congestive heart failure

The risk factors and biochemical markers associated with the

development of CHF in the general population are listed in

Table 2 Although no systematic investigation has been

performed to dissect the relative contribution of each of these

factors to the development of CHF in RA, several

well-defined contributing factors have been shown to be

over-represented in RA Whether the increased risk of CHF in RA

is primarily due to the effects of known risk factors, or to

unidentified risk factors unique to RA, is currently unknown,

though the predictors analyses by Wolfe et al [27] and

Nicola et al [26] (discussed above) suggest that both

traditional and RA-specific risk factors for CHF are operative

The available evidence on the prevalence of some of these

important risk factors for CHF in RA is reviewed below,

though it is important to recognize that RA is a heterogeneous disorder, and some factors may represent different risks for different subpopulations of RA patients

Hypertension

Systemic hypertension is one of the most potent risk factors for CHF, conferring a two- to three-fold increase in CHF risk for affected individuals [31] Chronic hypertension promotes the development of CHF by a variety of mechanisms, including the induction of maladaptive myocardial remodeling and atherosclerosis Reports of the prevalence of hypertension in RA have yielded varied results, with authors reporting lower [32], equivalent [26,33,34], or elevated [3,24,35] mean systolic and/or diastolic blood pressures in

RA patients compared to matched controls Importantly, although any history of hypertension was strongly associated

with prevalent CHF in the study by Wolfe et al [27] (odds

Table 2 Established risk factors and associative markers for the development of congestive heart failure

Shown to be comparatively

Clinical risk factors

Coronary atherosclerosis/myocardial infarction +++

Intrinsic pulmonary disease +(+) Sleep apnea/sleep-disordered breathing +(?)

Echocardiographic predictors Asymptomatic left ventricular enlargement + Increased left ventricular mass + Asymptomatic left ventricular systolic dysfunction (+)/–

Left ventricular diastolic dysfunction +++

Biochemical risk markers

Medications Non-steroidal anti-inflammatory drugs ++

Rare causes of CHF in the general population

CHF, congestive heart failure + evidence for increased prevalence;

–, no evidence for increased prevalence; +/–, evidence equivocal for increased prevalence; +(?), questionable/insufficient evidence for increased prevalence

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ratio 2.6 (95% CI 2.1-3.2)), Nicola et al [26] found no

association between hypertension and risk of incident CHF in

RA patients followed for a median of 11.8 years

Additionally, other factors over-represented in RA patients,

such as the chronic use of non-steroidal anti-inflammatory

drugs and corticosteroids, are both known to promote fluid

retention and elevate systemic blood pressure [36] The

independent effect of these agents on the development of

CHF in RA is complex, however, and has yet to be directly

investigated

Coronary atherosclerosis/myocardial infarction

Myocardial infarction (MI) is the most potent risk factor for

CHF, with a population-attributed risk for the development of

CHF in the Framingham cohort of 34% for men and 13% for

women [31] In most cases, other risk factors for CHF (e.g.,

hypertension, diabetes, and smoking) also contribute to the

pathogenesis of coronary atherosclerosis Importantly,

unrecognized and silent MI represents up to 25% of all

myocardial ischemic events [37] and subclinical

athero-sclerosis (with no history of MI) is also associated with an

increased risk for the development of CHF [38]

Several studies have confirmed an approximately two- to

four-fold increase in risk for MI among RA patients compared to

non-RA controls [3,5,34] Wolfe et al [27] showed that

recent MI (within six months) and any history of MI were both

significant univariate correlates of prevalent CHF in RA

patients (odds ratio 16.1 (95% CI 11.0-23.7) and 6.6 (95%

CI 5.4-8.0), respectively) In the study by Nicola et al [26],

ischemic heart disease (including overt MI, silent MI, and

angina) and risk factors for CVD accounted for the risk of

incident CHF in RF negative, but not RF positive, RA patients

Subclinical atherosclerosis, as measured by carotid

ultrasound, is more prevalent in RA patients compared to

matched controls [39] However, the relationship of

subclinical atherosclerosis to the risk of CHF (in the absence

of clinically recognized ischemic heart disease) in RA patients

is currently unknown

Diabetes

Although diabetes is a well-recognized risk factor for the

development of CHF [40,41], the prevalence of diabetes

does not appear to be increased in RA patients compared to

non-RA controls [24,42] Glucose intolerance/peripheral

insulin resistance has, however, been associated with an

increase in CHF risk in both cross-sectional [43] and

prospective, population-based [40] studies, and may be

increased in RA [44,45]

Valvular heart disease

Hemodynamically significant cardiac valvular disease may

lead to overt CHF through maladaptive compensatory

mechanisms resulting in myocardial remodeling (the

molecular basis of which is discussed below) Both necropsy [46] and cross-sectional echocardiographic studies [47] of

RA hearts have identified an increased prevalence of granulomatous and non-granulomatous valvular abnormalities, particularly of the mitral valve, and an increased prevalence of mitral regurgitation in RA patients compared to matched

non-RA controls No longitudinal echocardiographic studies have been performed, however, to determine the impact of this finding on the subsequent risk for developing CHF in affected

RA patients Destructive valvular lesions leading to complete valvular incompetence have been reported [48,49] but are considered rare occurrences

Intrinsic pulmonary disease

A wide spectrum of intrinsic pulmonary disorders, including disorders of the pulmonary air-spaces (chronic obstructive pulmonary disease), parenchyma (interstitial lung disease, pulmonary fibrosis), and vasculature (primary pulmonary hypertension) are associated with increasing pulmonary vascular resistance, progressive hypertrophy of the right ventricle, and eventual right heart failure with clinical CHF [50] In RA, pulmonary disease may be a manifestation of the

RA disease process itself or a result of RA-directed therapies (methotrexate, D-penicillamine, gold and others) [51] While symptomatic chronic pulmonary diseases are more prevalent

in RA patients compared with non-RA controls [24,52], subclinical pulmonary disease, including airways disease [53] (bronchiectasis, bronchiolitis) and parenchymal disease (interstitial pneumonitis), have been noted in nearly 50% of unselected RA patients in one series [54] In addition, several echocardiographic studies have suggested higher right ventricular systolic pressures in RA patients compared to

non-RA controls [55-57] In the study by Dawson et al [56],

pulmonary parenchymal disease could only account for 6% of

RA cases with increased pulmonary arterial pressures, suggesting that asymptomatic primary pulmonary vascular disease may be under-appreciated in RA These findings, and their putative effect on the subsequent development of CHF, warrant further study

Sleep apnea/sleep-disordered breathing

Although sleep apnea has been shown to be highly prevalent

in people with CHF in cross-sectional studies [58], no prospective population-based studies, to date, have investigated the putative effect of sleep apnea on the risk of CHF Sleep apnea is known, however, to increase systemic blood pressure via hypoxia-induced activation of the sympathetic nervous system [59], increase right ventricular pressure via hypoxia-induced pulmonary vasoconstriction [60], potentiate hypoxia-induced coronary ischemia [61], and induce the production of inflammatory cytokines such as IL-6 and tumor necrosis factor (TNF)-α [62], all recognized contributors to CHF risk Few studies of sleep apnea in RA exist, though the disorder has been reported in RA in the context of cervical spine and temporomandibular joint involvement [63] Recent reports of substantial improvements

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in sleep apnea-associated daytime somnolence in patients

treated with TNF inhibitors [64,65] suggests that the problem

may be under-appreciated in RA At present, however, any

link between sleep apnea and CHF risk in RA is speculative

Other factors

Smoking and obesity are established risk factors for both RA

[66] and CHF [41,67] Both are thought to promote CHF

primarily through exacerbation of atherogenesis, though both

may also potentiate the release of agents with direct toxicity to

the myocardium itself [68,69] Interestingly, although RA

patients may have similar or lower body mass indices than

non-RA counterparts, loss of skeletal muscle mass

accompanied by a compensatory increase in total fat mass in

RA patients may account for the stability of body mass indices

[70] Though no focused investigations have been undertaken

to date, this condition, termed sarcopenic obesity, could

predispose RA patients to higher than expected CHF risk

Rheumatoid arthritis associated factors

In general, myocardial nodules, restrictive pericarditis, and

coronary vasculitis are exceedingly rare causes of CHF [71];

however, older necropsy studies of RA hearts [32,46,72] have

indicated a higher prevalence of each of these complications

compared to the hearts of autopsied non-RA patients More

recent series using transthoracic echocardiography [55,56]

have identified a much lower prevalence of pericarditis than

that reported in the autopsy studies (2% versus 29% to 40%)

In a series using transesophageal echocardiography [47],

however, thirteen percent of RA patients were found to have

clinically silent pericarditis versus zero percent of non-RA

controls Case reports of rheumatoid nodules [73,74],

restrictive pericarditis [75,76], and coronary vasculitis [77] in

RA patients resulting in CHF are not uncommon in the

literature, although it is likely that these entities account for

only a small portion of the excess cases of CHF in RA

Echocardiographic predictors of congestive heart failure

Several large prospective studies have identified

asympto-matic left ventricular (LV) enlargement, hypertrophy and

dysfunction as significant risk factors for the development of

CHF [78-80] The strength of these associations, combined

with the documented efficacy of angiotensin converting

enzyme inhibitor therapy in delaying disease progression,

have prompted consensus recommendation of medical

treatment for these conditions classified as subclinical stages

of CHF [81] Importantly, once global alterations of LV

architecture and function are established, progression to

CHF with functional deterioration and eventually death is

inexorable [82] This unfavorable evolution highlights the

need to define earlier stages of myocardial dysfunction,

particularly in individuals with known risk factors for CHF

CHF with preserved systolic function

Between 30% and 50% of patients with CHF have preserved

systolic function (defined as LV ejection fraction (LVEF)

≥ 45-50%) [83,84] Despite the fact that this condition is associated with lower mortality when compared to heart failure with reduced LVEF, patients with CHF and normal LVEF have a four-fold increase in mortality relative to the normal population [83] In asymptomatic individuals, diastolic dysfunction with preserved systolic function is also predictive

of the subsequent development of overt CHF [85]

To date, a number of Doppler echocardiographic studies have been performed in RA patients without clinical evidence of CHF [55,86-94] (Table 3) Although limited by small numbers of patients and, in some cases, failure to provide a non-RA comparator group, these studies are consistent in demon-strating a high prevalence of asymptomatic diastolic dysfunction

in the setting of generally preserved systolic function A correlation between the degree of diastolic dysfunction and RA disease duration was shown in several investigations [92,94] Without longitudinal assessments, however, few conclusions can be made about the long-term effects of RA disease activity

on cardiac structure and function or, more importantly, factors influencing the transition from asymptomatic myocardial dysfunction to clinical CHF in RA

Impaired diastolic filling is felt to relate physiologically to impair-ment in relaxation or compliance of the left ventricle, resulting in elevated LV filling pressures and resultant elevated back pressures through the pulmonary circulation, right heart, and beyond [95] Histologically, processes that tend to stiffen the myocardium (e.g., hypertrophy, fibrosis, or infiltrative diseases)

or reduce compliance (e.g., restrictive pericarditis) can manifest

as diastolic dysfunction We postulate that chronic low-grade myocardial inflammation resulting in fibrosis may predispose patients with RA to diastolic dysfunction (discussed below)

The limitations of standard echocardiography, which include poor endocardial definition, lack of inter-observer reproducibility

of ejection fraction estimates, and lack of standardization of diagnostic criteria for diastolic dysfunction [96], often make it difficult to be precise about the diagnosis of diastolic dysfunction In practice, however, the diagnosis of diastolic CHF

is generally based on the finding of typical symptoms and signs

of CHF in a patient who is shown to have a normal LVEF and no valvular abnormalities on echocardiography [84] Newer noninvasive imaging methods, including contrast echocardio-graphy and cardiac magnetic resonance imaging, have been developed that permit greater precision and accuracy in the assessment of myocardial function Accordingly, the incorpora-tion of these newer imaging modalities into studies exploring CHF in RA may not only serve to improve diagnostic accuracy, but also provide predictive power and insights into the underlying pathophysiologic mechanisms of disease

Biomarkers associated with congestive heart failure risk

Cardiac natriuretic hormones

Recently, the measurement of circulating levels of brain natriuretic peptide has become available as a means of

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identifying patients with elevated LV filling pressures who are

likely to exhibit signs and symptoms of CHF Although the

role of cardiac natriuretic hormones in the identification and

management of individuals with asymptomatic ventricular

dysfunction remains to be fully clarified [97], elevated serum

levels of brain natriuretic peptide and amino-terminal pro-atrial

natriuretic peptide have been associated with an increased

risk of subsequent CHF in a community-based epidemiologic

study [98] In one small-scale cross-sectional study, RA

patients were found to have higher serum atrial natriuretic

peptide levels than healthy, non-RA controls [99] Another

cross-sectional study suggested that brain natriuretic peptide

levels may be elevated in RA patients independent of overt or

subclinical myocardial dysfunction [100] To date, no studies

have been performed in RA patients to establish the role of

cardiac natriuretic hormones in either risk stratification or

diagnosis of CHF

Hyperhomocysteinemia

Elevated serum levels of homocysteine have been

independently linked to an increased risk of CHF [101],

particularly in women [102] Hyperhomocysteinemia may

promote the development of CHF through induction of

atherosclerosis [103] and by direct effects on the

myo-cardium leading to myocardial remodeling [102] (discussed

below) In RA patients, homocysteine levels have been shown

to be significantly higher than those of matched non-RA controls [104] and are associated with both markers of inflammation and therapy with methotrexate [105] Though folic acid treatment reduces homocysteine levels in RA patients [105], and combination therapy with methotrexate and folic acid has been recently shown to be associated with

a reduced incidence of CVD in veterans with RA [106], the complex relationship of RA-induced and RA therapy-induced hyperhomocysteinemia to CHF risk in RA has yet to be completely elucidated

Inflammatory cytokines

In patients with overt CHF, levels of inflammatory cytokines (TNF-α, IL-6 and/or TNF-α receptors) are elevated and correlate with the severity of the disease [107-112] regard-less of etiology of CHF In patients with no overt CHF or history of ischemic heart disease, those with the highest serum levels of IL-6, C-reactive protein (CRP), and peripheral-blood mononuclear cell TNF-α were shown to have a two- to four-fold higher risk of developing CHF compared to patients with the lowest baseline levels of these cytokines [113,114]

In patients with overt CHF, both circulating peripheral-blood mononuclear cells and cells localized to the myocardium, including infiltrating inflammatory cells and cardiac myocytes,

Table 3

Doppler echocardiographic studies in patients with RA

Publication No of RA No of control Reference year patients subjects Findings (RA compared to control)

Mustonen et al [86] 1993 12 (males; 14 (males only; LV diastolic functional impairment

age 20-40 years) unmatched) No differences in LV systolic function

Corrao et al [88] 1996 40 40 non-RA Increased interventricular septal thickness

Increased LV mass index

LV diastolic functional impairment

Wislowska et al [89] 1998 100 100 non-RA Increased LV diastolic diameter

Reduced LV ejection fraction

Montecucco et al [90] 1999 54 54 non-RA Impaired diastolic relaxation

No differences in LV systolic function or LV diastolic diameter

Wislowska et al [91] 1999 35 with 35 with Increased valvular disease in nodular RA

nodular RA non-nodular RA Decreased LV ejection fraction in nodular RA

Di Franco et al [92] 2000 32 33 non-RA LV diastolic functional impairment

(unmatched) Positive correlation with RA disease duration (r = 0.40)

Cindas et al [87] 2002 40 48 non-RA LV diastolic functional impairment

Longer disease duration with more abnormal echocardiographic parameters noted

Alpaslan et al [93] 2003 32 with long 32 non-RA LV diastolic functional impairment

standing RA (unmatched) Normal systolic function in all

Levendoglu et al [94] 2003 40 – LV diastolic functional impairment

Positive correlation with RA disease duration Gonzalez-Juanatey 2004 47 treated 47 LV diastolic functional impairment

et al [55] RA patients Positive correlation with extra-articular manifestations of RA

LV, left ventricular; RA, rheumatoid arthritis

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have been shown to be the source of the elevated cytokine

levels [112,115,116]

In the inflamed synovium of the rheumatoid joint,

macrophage-derived cytokines such as TNF-α, IL-1 and IL-6

are prominently expressed, and inhibitors of these cytokines,

particularly TNF inhibitors, have been proven to be highly

successful therapies for RA [117-120] In RA, the inflamed

synovium as well as peripheral-blood mononuclear cells

contribute to elevated circulating TNF-α (and TNF receptor)

levels To date, however, the potential contribution of the

myocardium in RA as a source of local cytokine production

has not been investigated

Other conditions associated with chronically elevated levels

of inflammatory cytokines (e.g., aging, chronic kidney disease,

obesity) are also associated with an increased prevalence of

CHF [121,70]; however, as in RA, the contributions of

various non-inflammatory confounders in each of these

conditions to the pathogenesis of CHF have not been fully

explored

Clinical studies of TNF inhibitors and

congestive heart failure

The potent association of inflammatory cytokines with both

CHF risk and clinical worsening of existing CHF has

prompted speculation that pharmacologic cytokine inhibition

might prove an effective treatment for established

symptomatic CHF and/or reduce the risk of developing CHF

in patients who are potentially at risk for CHF secondary to

chronic cytokine excess (i.e., patients with RA and other

chronic systemic inflammatory disorders) The unfavorable

and unanticipated results of clinical trials investigating the

use of anti-TNF-α therapy to treat advanced CHF, however,

have raised concerns that TNF inhibitors may actually be

harmful to the myocardium To address this apparent

contradiction we next examine the conflicting human clinical

experience relating TNF inhibitors to CHF in the context of

the available animal data on cytokine-induced CHF

Use of TNF inhibitors as a treatment for advanced

congestive heart failure

Both etanercept, a soluble decoy TNF receptor, and

infliximab, a chimeric anti-TNF-α monoclonal antibody, have

undergone efficacy and safety evaluations in multicenter,

double-blind, placebo-controlled trials for the treatment of

patients with advanced symptomatic CHF [122,123] The

study designs of these trials (‘RENNAISSANCE’

(Randomized Etanercept North American Strategy To Study

Antagonism Of Cytokines) and ‘RECOVER’ (Research Into

Etanercept: Cytokine Antagonism In Ventricular Dysfunction

Trial) [123] for etanercept, and ‘ATTACH’ (Anti-TNF-α

Therapy Against Chronic Heart Failure) [122] for infliximab)

have been recently reviewed in detail [124] The collective

results of the trials were generally unfavorable, with

RENNAISSANCE and RECOVER halted in June 2001 when

an interim analysis revealed that continuation would be highly unlikely to show a statistically significant difference in outcomes between the treatment groups [123] and ATTACH demonstrating no clinical efficacy of infliximab, but higher rates of hospitalizations and all-cause mortality in patients treated with the highest dose (10 mg/kg) of infliximab compared to placebo [122]

The high-profile and well-publicized nature of these trials, coupled with a 2002 report, using the US Food and Drug Administration’s MedWatch post-licensure database for voluntary reporting of adverse events, of new or worsening CHF in 47 of approximately 300,000 patients worldwide treated with infliximab or etanercept (of whom 38 (81%) had

no prior history of CHF, and 10 of whom were less than

50 years of age) have led some to conclude that TNF inhibition may exert a detrimental, rather than protective, effect on the myocardium of RA patients To date, the only available evidence to refute this supposition comes from

Wolfe et al [27], in which a statistically significant lower rate

of self-reported, physician-diagnosed CHF was determined in

RA patients receiving treatment with a TNF inhibitor compared to those not treated with TNF inhibitors, even after adjustment for unbalanced clinical characteristics and previous history of CVD (2.8% versus 3.9%, respectively,

P = 0.03) A lower rate of incident CHF in TNF inhibitor

treated versus untreated patients was also demonstrated (3.5% versus 4.3%, respectively) when the analysis was limited only to data collected after the Food and Drug Administration warning following the RENNAISANCE, RECOVER, and ATTACH trials, although this difference was not statistically significant No cases of incident CHF in TNF inhibitor treated RA patients who were less than 50 years of age were found, although three cases of incident CHF were reported in RA patients under 50 years of age who were not treated with TNF inhibitors As noted above, the diagnosis of CHF in this study was not based on predefined clinical and/or imaging criteria nor was the etiology of CHF (ischemic versus non-ischemic versus other) determined; nonetheless, this study provides tantalizing circumstantial support for the notion that TNF-α contributes to the etiology of CHF in RA Additional indirect support derives from a recent report [125]

in which the prescription of disease modifying antirheumatic drugs (DMARDs; including TNF inhibitors) was associated with a 30% reduction in hospitalizations for new-onset CHF from a large administrative claims database of RA patients Considering only TNF inhibitor treated patients, a 50% reduction in CHF hospitalizations was observed

To date, few human investigations into the direct effects of TNF-α or TNF inhibitors on the myocardium have been under-taken Imaging substudies of non-RA patients with advanced CHF showed no effect of etanercept on LVEF (assessed in

215 subjects who underwent radionuclide ventriculography

at baseline and at 24 weeks) in RENNAISANCE [126] and a modest increase in LVEF (measured by radionuclide

Trang 8

ventriculography) despite clinical worsening in infliximab

treated patients in ATTACH [122] Although studies

incorporating direct visualization of myocardial function have

yet to be performed in RA patients, clues from animal models

of CHF induced by chronic cytokine excess (a setting that

may mimic the RA disease state) may serve to explain the

apparent contradictions in treatment effects of cytokine

inhibition on the myocardium

Animal models of cytokine induced

congestive heart failure

In vitro and animal studies strongly support a mechanistic

role for macrophage-derived cytokines, especially TNF-α, in

the pathogenesis of CHF, rather than a mere

epipheno-menon Key features of the CHF phenotype, including

pulmonary edema, negative inotropy, ventricular dilatation and

hypertrophy, endothelial dysfunction, reduced myocardial

β-adrenergic responsiveness, and myocyte apoptosis are

recapitulated by experimental augmentation of TNF-α

[127-129] In a rat model, continuous infusion of TNF-α via an

implanted osmotic infusion pump to levels congruent with

those found in human CHF, led to a time-dependent

reduction in LVEF and development of left ventricular

dilatation [130] These effects were reversed, at least in part,

by removal of the infusion pump or administration of a dimeric

TNF receptor antagonist [130]

Transgenic murine models of cardiac-restricted

over-expression of TNF-α have been generated by coupling the

murine TNF-α gene to the murine α-myosin heavy chain

promoter [131-133] When expression is extremely robust,

the animals die quickly (mean 11 days) of a dilated

cardio-myopathy that, on histologic examination, is due to a diffuse

inflammatory myocarditis [131] With less robust expression

of TNF-α, survival is longer (mean time to death approximately

10 months) and the CHF phenotype evolves more gradually,

characterized by ventricular hypertrophy and dilatation,

interstitial infiltrates and fibrosis, and depressed adrenergic

response These effects were attenuated or blocked by

antagonism of TNF-α [134-136]

These studies strongly support a central role for TNF-α in

mediating the processes leading to myocardial dysfunction

An inflammatory myocarditis has also been described in

autopsy studies of RA patients (discussed above) It is

tempting to speculate that chronic production of cytokines,

including TNF-α, may affect the myocardium in RA in either an

endocrine (originating in the synovium) or paracrine (produced

in the local environment of the progressively failing

myocardium) fashion, contributing to subclinical and eventually

clinically recognizable ventricular dysfunction (Fig 1)

Inflammatory pathways and myocardial remodeling

The process by which cardiac structure and function adapts

to physiologic changes is termed ‘myocardial remodeling’ and

involves the cellular and interstitial changes leading to

myocyte hypertrophy, ventricular dilatation, alterations in interstitial collagen superstructure, and interstitial myocardial fibrosis [137] This process is mediated primarily through local expression of matrix metalloproteinases (MMPs), particularly MMP-1, MMP-2, MMP-3, and MMP-9, and modulated by expression of tissue inhibitors of matrix metalloproteinases (TIMPs) [138] Circulating levels of MMPs are elevated in patients with overt CHF, regardless of etiology [139-141], suggesting a common unifying mechanism Overexpression of MMPs and/or reduced expression of TIMPs are associated with proteolysis of the myocardial extracellular fibrillar collagen matrix and progressive ventricular dilatation [142,143] Selective and non-selective MMP inhibition reverses or blocks the development of the phenotype [144,145]

TNF-α has been shown to be a key regulator of MMP expression in myocardial remodeling [135,146] In transgenic mice with cardiac restricted overexpression of TNF-α [146], early exposure to elevated TNF-α was associated with an increase in the myocardial zymographic MMP activity/ myocardial TIMP (MMP/TIMP) ratio favoring degradation of interstitial fibrillar collagen and development of ventricular dilation and CHF With aging, however, a shift to increased myocardial TIMP levels and an overall reduction in the MMP/

Figure 1

Proposed pathogenesis of myocardial dysfunction in rheumatoid arthritis CRP, C reactive protein; DMARD, disease modifying anti-rheumatic drug; IL, interleukin; MMP, matrix metalloproteinase; NSAID, non-steroidal anti-inflammatory drugs; TNF, tumor necrosis factor

Trang 9

TIMP ratio, an increase in collagen production, and

subsequent fibrosis of the dilated ventricle was observed

This later phase was associated with an increase in

transforming growth factor-β expression [146] This

time-dependent effect of TNF-α induced myocardial remodeling

suggests that there may be a window of opportunity early in

disease during which events leading to myocardial interstitial

fibrosis may be prevented [147] Importantly, it is this shift

from myocardial interstitial degradation to fibrosis that

appears to play a key role in the transition from compensated

to decompensated CHF [148] Moreover, this is supported

by the finding that delayed anti-TNF-α therapy, administered

at six weeks of age, was able to reverse ventricular dilatation,

but not established fibrosis, in a transgenic mouse model of

cardiac TNF-α overexpression [149] The possibility that

enhancing TNF-α expression in late CHF might even be

desirable in order to reestablish a favorable MMP/TIMP

balance has not been explored

Recent work suggests that cardiac structural homeostasis is

regulated in part through a balance between

membrane-bound and cleaved TNF-α Normally, membrane membrane-bound

TNF-α is converted to its soluble form by cleavage with

TNF-α converting enzyme (TACE) [150] In a line of

transgenic mice with cardiac-restricted overexpression of

TNF-α that is resistant to cleavage by TACE, concentric

hypertrophy without chamber dilatation was observed

[151,152], whereas mice with overexpression of TNF-α and

an intact TACE cleavage site exhibited extracellular matrix

degradation and ventricular dilatation [151,152] In humans,

increased TACE expression parallels the increase in TNF-α

expression associated with dilated cardiomyopathy [153] and

myocarditis [154] Little is currently known, however, about

the relative amounts or contribution of membrane bound

TNF-α to myocardial homeostasis in humans

In summary, animal studies have shown that the processes

leading to cardiac myocyte hypertrophy, interstitial fibrillar

collagen degradation, mural realignment, and ultimately to

dilated cardiomyopathy are induced and/or regulated, at least

in part, by TNF-α The effects of TNF-α on the myocardium

are complex, however, with a pathogenic effect early on and a

putative protective effect later in disease This dichotomy has

important potential implications for human disease and has

preliminary support from available clinical studies of TNF

inhibitors in humans, in which patients with advanced CHF

have shown no benefit or worsened when treated with

anti-TNF-α therapy In contrast, in patients with RA and no overt

CHF, treatment of RA with TNF inhibitors might offer some

protection against cytokine induced CHF

Conclusions

The evolution of subclinical myocardial dysfunction to overt

CHF is associated with significant morbidity and alarming

mortality Population- and clinic-based epidemiologic studies

have suggested that RA patients may be more prone to the

development of CHF and more susceptible to CHF-related mortality While some traditional risk factors for CHF are over-represented in RA patients, they do not appear to account for all of the increased CHF risk observed Other RA associated factors, particularly the chronic elaboration of inflammatory cytokines, are likely substantial contributors to myocardial dysfunction in RA patients Additional investigation is needed

to clarify both the direct effects of the RA disease process and the effects of RA-directed therapeutics on the myocardium at all stages of disease in order to define appropriate strategies to prevent or attenuate the development of CHF in RA patients

Competing interests

Dr Giles receives grant support from the American College of Rheumatology and the Arthritis National Research Foundation

Dr Bathon receives grant support from the National Institutes

of Health, Bristol Myers Squibb, Centocor, Amgen, IDEC/Genentech Dr Bathon is a consultant to Abbott

Acknowledgments

This work was supported in part by an American College of Rheumatol-ogy Clinical Investigator Fellowship Award (JTG), an Arthritis National Research Foundation Award (JTG), and by Grant AR050026-01 from the National Institute of Arthritis and Musculoskeletal and Skin Dis-eases (JMB)

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